Teo Ada E D, Garg Sumedha, Shaikh Lalarukh Haris, Zhou Junhua, Karet Frankl Fiona E, Gurnell Mark, Happerfield Lisa, Marker Alison, Bienz Mariann, Azizan Elena A B, Brown Morris J
From the Clinical Pharmacology Unit, Centre for Clinical Investigation, Addenbrooke's Hospital, University of Cambridge (A.E.D.T., S.G., L.H.S., J.Z., E.A.B.A., M.J.B.), Department of Medical Genetics, Division of Renal Medicine, University of Cambridge (F.E.K.F.), University of Cambridge Metabolic Research Laboratories, Wellcome Trust-Medical Research Council (MRC) Institute of Metabolic Science, Addenbrooke's Hospital (M.G.), Department of Pathology, Addenbrooke's Hospital (L.H., A.M.), and MRC Laboratory of Molecular Biology (M.B.) - all in Cambridge, United Kingdom; and the Department of Medicine, National University of Malaysia Medical Centre, Kuala Lumpur (E.A.B.A.).
N Engl J Med. 2015 Oct 8;373(15):1429-36. doi: 10.1056/NEJMoa1504869. Epub 2015 Sep 23.
Recent discoveries of somatic mutations permit the recognition of subtypes of aldosterone-producing adenomas with distinct clinical presentations and pathological features. Here we describe three women with hyperaldosteronism, two who presented in pregnancy and one who presented after menopause. Their aldosterone-producing adenomas harbored activating mutations of CTNNB1, encoding β-catenin in the Wnt cell-differentiation pathway, and expressed LHCGR and GNRHR, encoding gonadal receptors, at levels that were more than 100 times as high as the levels in other aldosterone-producing adenomas. The mutations stimulate Wnt activation and cause adrenocortical cells to de-differentiate toward their common adrenal-gonadal precursor cell type. (Funded by grants from the National Institute for Health Research Cambridge Biomedical Research Centre and others.).
近期对体细胞突变的发现使得能够识别出具有不同临床表现和病理特征的醛固酮瘤亚型。在此,我们描述了三名患有醛固酮增多症的女性,其中两名在孕期发病,一名在绝经后发病。她们的醛固酮瘤携带CTNNB1激活突变,该基因在Wnt细胞分化途径中编码β-连环蛋白,并且表达编码性腺受体的LHCGR和GNRHR,其水平比其他醛固酮瘤中的水平高出100倍以上。这些突变刺激Wnt激活,并导致肾上腺皮质细胞向其共同的肾上腺-性腺前体细胞类型去分化。(由英国国家卫生研究院剑桥生物医学研究中心等机构的资助。)